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Journal of Pediatric Urology (2013) xx, 1e4

A novel alternative for renal replacementtherapy: 2-year successful colonic dialysisvia a Malone antegrade continent enemastoma

Abdol-Mohammad Kajbafzadeh a,*, Atefeh Zeinoddini a,Reza Heidari a, Haleh NaserHodjjati a, Ali Tourchi b

a Pediatric Urology Research Center, Pediatric Center of Excellence, Tehran University of MedicalSciences, Tehran, Islamic Republic of IranbDivision of Pediatric Urology, Brady Urological Institute, The Johns Hopkins Medical Institutions,Baltimore, MD, USA

Received 24 April 2013; accepted 13 November 2013

KEYWORDSDialysis;End stage renaldisease;Intestinal/colonicdialysis;Renal replacementtherapy

* Corresponding author. 2nd Floor, NAve., Tehran 1998714616, Islamic Re2208 9946; fax: þ98 21 2206 9451.

E-mail address: kajbafzd@sina.tum

Please cite this article in press as: Kadialysis via a Malone antegradej.jpurol.2013.11.009

1477-5131/$36 ª 2013 Journal of Pedhttp://dx.doi.org/10.1016/j.jpurol.20

Abstract This study is a case report of home-based colonic dialysis (CD) for treating end-stage renal disease in a 20-year-old woman. She had a history of Malone antegrade continenceenema (MACE) for treating neuropathic bowel at the age of 11 years. The patient refused anytype of renal replacement therapy. However, she agreed to CD through the MACE stoma bychanging the colonic irrigation solution to the peritoneal dialysis solution. The patient was dis-charged with a plasma creatinine (Cr) level of 1.7 mg/dL and blood urea nitrogen (BUN) level of8 mg/dL. She has continued CD on a regular basis at home. The patient’s serum Cr and BUN hasremained in the steady low state during 24 months of follow-up (mean Cr level Z 2.8 mg/dLand mean BUN level Z 10.7 mg/dL).ª 2013 Journal of Pediatric Urology Company. Published by Elsevier Ltd. All rights reserved.

o. 32, 7th Street Saadat-Abadpublic of Iran. Tel.: þ98 21

s.ac.ir (A.-M. Kajbafzadeh).

jbafzadeh A-M, et al., A novel altcontinent enema stoma, Jour

iatric Urology Company. Publishe13.11.009

Introduction

Considering the high annual cost and morbidity of dialysisand unavailability of these facilities in some countries[1,2], finding an alternative treatment for renal replace-ment therapy (RRT) is inevitable. Following our ongoingexperimental intestinal dialysis in an animal model (whichwill be published soon), we used colonic dialysis (CD)

ernative for renal replacement therapy: 2-year successful colonicnal of Pediatric Urology (2013), http://dx.doi.org/10.1016/

d by Elsevier Ltd. All rights reserved.

2 A.-M. Kajbafzadeh et al.

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through a Malone antegrade continence enema (MACE)stoma as a simple, reproducible alternative treatment forRRT. This study is a case report using home-based CD intreating end-stage renal disease (ESRD) in a 20-year-oldwoman who had rejected any kind of dialysis.

Case report

In 2010, a 20-year-old woman was admitted to our centerdue to a rise in the serum creatinine level and high bloodpressure, and complaining of severe headache. At the ageof 11 years, the patient had undergone augmentationileocystoplasty with simultaneous MACE procedure andMitrofanoff principle, by dividing the long appendix withpreservation of two pedicles as previously described [3], forthe management of refractory urinary incontinence andsevere constipation in the setting of sacral agenesis. Thepatient was continent, using clean intermittent catheteri-zation (CIC). At admission, the patient’s blood pressure was190/110 mmHg and blood biochemistries were as the fol-lows: Cr, 6.5 mg/dL; blood urea nitrogen (BUN), 29 mg/dL;potassium, 3.1 mEq/L; sodium, 135 mEq/L; phosphorous,3 mg/dL; calcium, 8 mg/dL. There were no signs of acuterenal failure or urinary tract infection.

The patient refused any type of RRT. However, sheagreed to intestinal (colonic) dialysis by perfusing standardperitoneal dialysis (PD) solution through the MACE stoma;therefore, CD was started through the patient’s MACEstoma (PD solution: sodium, 132 mEq/L; calcium, 3.5 mEq/L; chloride, 1.5 mEq/L; lactate, 35 mEq/L; glucose, 1.5%).The patient’s appendicocecostomy was intubated with asilicon catheter. We perfused the standard PD solution;however, regarding our previous animal experience withintestinal dialysis, we supplemented the PD solution withsome additives in order to increase the efficacy of thistechnique. Ten grams of activated charcoal, which wasdissolved in 20 mL of dialyzate, was added to the PD solu-tion. Activated charcoal increases the virtual colonic sur-faces; by binding waste products to the activated charcoal,the concentration gradient between blood and the intesti-nal tract can be maintained for continuous diffusion ofwaste products, which can cause significant reduction inserum BUN and Cr level [4]. One hundred milliliters of 10%mannitol was also added to 2 L of PD solution. We hy-pothesized that the osmotic effect of mannitol besidesglucose in dialyzate would help for removal of excess

Figure 1 (A) The patient’s appendicocecostomy is intubated andview of intestinal dialysis. (C) The patient is ambulatory while the

Please cite this article in press as: Kajbafzadeh A-M, et al., A novel altdialysis via a Malone antegrade continent enema stoma, Jouj.jpurol.2013.11.009

water. The patient was trained to dialyze herself and wasdischarged after 2 weeks with normal blood pressure (115/80 mmHg), serum creatinine level of 1.7 mg/dL, BUN levelof 8 mg/dL, and stable biochemical parameters despitemild hypocalcemia. The patient was discharged on ami-loride [5 mg 3 times a day], furosemide [40 mg twice a day],hydralazine [50 mg 4 times a day], enalapril [5 mg 3 times aday], losartan [25 mg twice a day], oxybutynin [2.5 mg 3times a day], folic acid, B complex, ferrous sulfate, vitaminB6 and vitamin E. She continued CD, usually on a regularbasis at home, with two or three cycles per day, and in eachcycle she perfused 2 L of PD solution in about 3 h; however,there were some episodes of discontinuation of CD duringthe 2-year follow-up period (Fig. 1A,B). The patient ismobile and able to do physical activities during intestinalperfusion (Fig. 1C). It is noteworthy that the patient nolonger suffered from constipation after starting CD, as CDworks as a colonic washout too. She defecates continentlywithout difficulty once or twice a day. The patient per-formed CIC as in previous years and reports excretion ofabout 5 L of urine daily through CIC. During the 24-monthfollow-up, serum BUN and Cr levels remained at a consis-tent low level (mean Cr level, 2.8 mg/dL; mean BUN level,10.7 mg/dL) (Fig. 2A,B). Serum concentration of sodium,potassium, and phosphorous remained within the normalrange except for two episodes of hypokalemia (K Z 3.3 and3.1 mEq/L); as the hypokalemia was not severe it wasmanaged by oral administration of potassium chloride(KCL). The patient’s serum Cr level rose to 6.3 and 6.8 mg/dL temporarily when she stopped CD for a short time of herown record. These episodes were managed subsequently byCD (Fig. 2A,B).The patient received renal transplantationfrom lining non-related donor 2-month ago and he serumcreatinine level is normal (1 mg/dl) and urine output of2500 ml/24 h with controlled hypertension with minimalanti-hypertensive medications. The MACE channel stoma isirrigated by tap water for management of constipation.

Discussion

The present case represents the efficacy of MACE stomadialysis in a 20-year-old woman with ESRD who had under-gone the MACE procedure previously due to neuropathicbladder and bowel.

The MACE procedure has proved an effective treatmentfor constipation and fecal incontinence in patients

the dialyzate is infused by gravity drainage. (B) The schematicintestinal dialysis is being performed.

ernative for renal replacement therapy: 2-year successful colonicrnal of Pediatric Urology (2013), http://dx.doi.org/10.1016/

Figure 2 (A) The serum bloodeureaenitrogen (BUN) level (mg/dL) at 24-month follow-up; the second and third peaks indicatewhen the patient stopped CD for a week. (B) The serum creatinine level (mg/dL) at the 24-month follow-up; the second and thirdpeaks indicate when the patient stopped colonic dialysis (CD) for about a week.

A novel alternative for renal replacement therapy 3

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unresponsive to conservative measures [5,6]. It is widelyused in various disorders, including spina bifida, myelome-ningocele, anorectal malformations, and idiopathic andfunctional constipation [5,6]. Most of mentioned anomaliesare accompanied by concomitant urological problems andneurogenic bladder, which may result in voiding impair-ment, urine stasis, and vesicoureteral reflux (VUR) togetherwith recurrent pyelonephritis, which can damage the kid-neys and progress to ESRD. The patients who develop toESRD need renal replacement therapy. In patients withconcomitant neuropathic bowel and bladder who undergoan ACE procedure for the management of neuropathicbowel and concurrently suffer from ESRD, the intestinalsurface area can act as a semi-permeable membrane suchthat small soluble particles, like creatinine, urea, and otherwaste products, can diffuse from plasma into the gastro-intestinal tract [7,8]. This technique can be done easily byperfusing dialyzate solution through an ACE stoma. Tech-niques of intestinal perfusion have been performed by usingvarious methods; these techniques involve the intestinalmucosa surface as a semi-permeable membrane for diffu-sion of waste metabolic products from circulating blood tothe intestinal tract. Schloerb [8] used isolated jejunumsegment perfusion to manage a patient with advanceduremia; it was found to be a useful method in maintenanceof water and electrolyte homeostasis and removingnitrogenous metabolic wastes. Taguchi et al. [9] noted thefeasibility of Roux-en-Y anastomosis at two sites, theproximal and distal of jejunum, as an alternative approachfor removing metabolic wastes. Rosin [7] assessed thesuccess of isolated colonic loop perfusion primarily in thesheep model of renal failure and subsequently in human.Confirming our findings, he concluded that this techniquecould sufficiently remove toxic metabolites, maintainelectrolyte homeostasis, and preserve water balance.However, May et al. [10], in their study on bilaterallynephrectomized miniature pig, reported very little clinicalvalue for intestinal dialysis for removal of toxins throughthe small and the large intestine. One may argue that there

Please cite this article in press as: Kajbafzadeh A-M, et al., A novel altdialysis via a Malone antegrade continent enema stoma, Jourj.jpurol.2013.11.009

is a paucity of knowledge in the literature regarding theapplicability of intestinal dialysis in human.

In our experience of dialysis through the MACE stoma,the patient suffered no adverse gastroenterological sideeffects, and no hemodialysis or PD was needed. Moreover,it is noteworthy that patients with PD have a high risk ofinfection, either at the catheter site or at the abdominalcavity (peritonitis), due to direct access to the peritonealcavity [11]. Other complications of PD include abdominalhernia, leaking around the catheter, and encapsulatingperitoneal sclerosis [12,13]. In the present technique thereis no risk of infection and fibrosis as the dialysis is per-formed through a previously fixated stoma; therefore, thismakes CD a good alternative to PD when conventionalmethods are not suitable in patients who suffer fromrecurrent peritonitis following PD. Furthermore, the startand annual costs of this procedure are inexpensive andallow the patient to perform dialysis easily at home orwhere no hospital facilities or trained staff are accessible.The annual cost of continuous ambulatory peritoneal dial-ysis (CAPD) in Iran is US$10,674, and is completely sup-ported by the government budget. The major cost of CAPDin Iran is related to laparoscopic insertion of a CAPD cath-eter and the single-use dialysis tubing line used for peri-toneal dialysis bags. The annual cost for this simplemodality (CD) is approximately one-quarter that of theCAPD annual cost (US$2555). However, the annual cost ofCAPD in Western countries is between US$22,759 andUS$37,000.

Needless to say, the long-term clearance obtained bythis approach is speculative at best, and long-term clinicaltrials are needed. Finally, the authors believe that CDthrough a MACE stoma may be a valuable option whenconventional methods are not available.

Conflict of interest

None.

ernative for renal replacement therapy: 2-year successful colonicnal of Pediatric Urology (2013), http://dx.doi.org/10.1016/

4 A.-M. Kajbafzadeh et al.

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Funding

None.

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